Ten corneal specimens obtained at the time of penetrating keratoplasty for long-standing refractory Acanthamoeba keratitis were reviewed. Histopathologic findings included epithelial denudation and variable degrees of necrosis, inflammation, and cysts or trophozoites of acanthamoeba in the stroma. No blood vessels were found in the stroma despite the presence of long-standing corneal inflammation. The absence of corneal stromal neovascularization on biomicroscopic and histopathologic examination is a sign of Acanthamoeba keratitis.